Int. J. Oral Maxillofac. Surg.

2014; 43: 1171–1175

http://dx.doi.org/10.1016/j.ijom.2014.05.011, available online at http://www.sciencedirect.com

Clinical Study
Craniofacial Surgery

Preclinical pathways to S. Klubaa, J. Lypkea, W. Krauta,

M. Krimmela, K. Haas-Ludeb,
S. Reinerta

treatment in infants with

a
Department of Oral and Maxillofacial
Surgery, University Hospital Tübingen,
Germany; bUniversity Children’s Hospital,
Department of Paediatric Neurology,

positional cranial deformity Tübingen, Germany

S. Kluba, J. Lypke, W. Kraut, M. Krimmel, K. Haas-Lude, S. Reinert: Preclinical

pathways to treatment in infants with positional cranial deformity. Int. J. Oral
Maxillofac. Surg. 2014; 43: 1171–1175. # 2014 International Association of Oral and
Maxillofacial Surgeons. Published by Elsevier Ltd. All rights reserved.

Abstract. Positional plagiocephaly in infants is frequent. As well as positioning,

physiotherapy, and osteopathy, helmet therapy is an effective treatment option. The
outcome also depends on the timely initiation of treatment. We investigated the
preclinical pathways to treatment. Parents of 218 affected children were
interviewed. Data were collected regarding detection and the treatments used prior
to the first craniofacial consultation at the study clinic in Germany. Descriptive and
statistical analyses were performed. For 78.4% of the children, the cranial
deformities were first detected at 4 months of age. One hundred and twenty-two
children received helmet therapy. Parents consulted the paediatrician with a mean
latency of 0.4 months; 3.3 months passed until the first craniofacial consultation.
Approximately 90% were treated with repositioning and 75.2% received additional
physiotherapy or osteopathy prior to presentation. Children treated with
physiotherapy/osteopathy presented significantly later (P = 0.023). The time lapse
to craniofacial consultation was not significantly different between children with
Keywords: positional plagiocephaly; brachyce-
and without later helmet therapy. We identified a relevant delay between the
phaly; helmet therapy; treatment pathways;
detection of positional cranial deformity and consultation with a craniofacial treatment delay.
specialist. For affected children, this may potentially compromise the outcome of
helmet therapy. Early referral to a specialist and if necessary the simultaneous Accepted for publication 14 May 2014
application of different treatments should be preferred. Available online 15 July 2014

Positional skull deformities (plagioce-
phaly and brachycephaly) have increas-
ingly become a focus of medical interest
over the last two decades. As a result of
clinical demand we established a special-
ist clinic a few years ago.
Typical clinical signs are a parallelo-
gram-style sloping head shape (plagioce-
phaly) or an abnormal head width to head
length ratio (brachycephaly). A combina-
tion of both types is common. Skull de-
formities resulting from premature closure
of the cranial sutures (craniosynostosis),
especially lambdoid suture synostosis, can
appear to be clinically very similar. They
can be distinguished from one another by
cranial suture ultrasound,1 and are usually
treated surgically.
The treatment of plagiocephaly and
brachycephaly is usually interdisciplinary
and interprofessional. The therapeutic
spectrum ranges from waiting for a spon-
taneous improvement and positioning
methods, to physiotherapy and osteopa-
thy, and then to the much-discussed
helmet therapy. Helmet therapy regulates
the head shape by controlling growth in

0901-5027/01001171 + 05 # 2014 International Association of Oral and Maxillofacial Surgeons. Published by Elsevier Ltd. All rights reserved.
1172 Kluba et al.
Fig. 1. Infant with positional plagiocephaly before and at the end of helmet therapy.
the deficient direction. The basic principle
is the enormous growth potential of the
skull, especially in the first year of life.2 A
harmonization, and ideally even complete
normalization of the head shape, is
achieved within a few weeks to several
months (Fig. 1). It is a low-risk, non-
invasive treatment; however it requires
the helmet to be worn consistently for
23 h a day if possible (Fig. 2). The treat-
ment has been investigated in numerous
studies and its efficacy is evident.3–13
However, because of growth dynamics,
the outcome depends on the timely initia-
tion of treatment.6,14–16 Several studies
Fig. 2. Child with a helmet.
have shown a significantly worse treat-
ment outcome when the treatment is
started after 6 months of age.6,14,15
While there is wide agreement on the
question of the aetiology among experts,
the long-term relevance of the problem
and the corresponding therapeutic strate-
gies, especially helmet treatment, are rated
very differently. There is also disagree-
ment regarding the financing of helmet
therapy, which in Germany is not yet
included on the health insurance compa-
nies’ lists of services.
These controversies cause noticeable
uncertainty for parents and persons re-
sponsible for the care and custody of the
child. Parents are often confronted with
conflicting opinions from different specia-
lists. This can result in the parents being
unable to cope with making a decision for
or against a therapeutic treatment, as they
no longer know what is best for their child.
In view of these facts and the time
constraints for the most optimal helmet
therapy result, the aim of this study was to
examine causes of delay in patient referral
to a skull deformity clinic. Prior to this
study we were unaware of the exact pre-
clinical pathways and how time-consum-
ing they are objectively.
Materials and methods
The study was performed using a tele-
phone survey of the parents or carers of
affected children attending our clinic for
positioning-related skull deformities.
Clinical data were also included in the
analyses. The data collection included
only children with positional cranial
deformities, with and without helmet
treatment. Exclusion criteria were prema-
ture closure of the cranial sutures or
unclear diagnosis. No child underwent
surgery.
The diagnosis was normally made di-
rectly at first presentation to our clinic.
Each child underwent an ultrasound ex-
amination prior to treatment in order
to exclude a craniosynostosis. In cases
with an indication for helmet therapy,
individual helmets were made by
Cranioform (Siegen, Germany) within
2 weeks. At the second appointment 2
weeks later, the fit of the helmet was
checked and parents were instructed on
the correct application and cleaning of
the helmet.
Basic data including gender and the
type and severity of the deformity were
recorded for each patient. The head diam-
eter, head length, and angles between the
skull diagonals were measured at the first
appointment in the clinic using a crani-
ometer. To quantify the degree of severity
of the deformity, the individual asymme-
try index of the skull, according to Love-
day and de Chalain,17 was calculated using
these data (cranial vault asymmetry index;
CVAI in %). A value of 0% indicated a
completely symmetrical head shape;
values >3.5% were considered to be path-
ological. The head width to head length
ratio (cranial index; CI in %) was also
calculated for brachycephaly. A value of
85% was deemed to be normal and values
>93% were considered to be very con-
spicuous; when the index is >100%, the
head is broader than long.
Using a questionnaire, standardized in-
formation about the initial recognition of
the conspicuous head shape and the path-
way to treatment was collected by tele-
phone survey (Fig. 3). We wanted to
identify the people involved, the methods
that had been used, and the prior chrono-
logical patterns and courses of events.
Depending on the question, single choice
or categorized answers were possible.
Data evaluation was performed using
IBM SPSS version 20.0 statistical soft-
ware (IBM Corp., Armonk, NY, USA).
The intervals between the various steps of
the care pathways were also recorded from
the collected data to quantify the time
delays.
Statistical group comparisons were per-
formed, as well as descriptive analyses.
A level of significance of a = 5% was
assumed, and a P-value of <0.05 was
defined as statistically significant. As the
Shapiro–Wilk test did not always show a
normal distribution, the Mann–Whitney
U-test was used. For the same reason,
the median value was also given in the

Fig. 3. General structure of the questionnaire.
descriptive analysis alongside the mean
value.
Results
Basic data
A total of 218 children were included in
the analysis. Forty-four percent (n = 96)
did not have a helmet; the other 56%
(n = 122) were treated with a helmet. Sev-
enty percent (n = 152) of the affected chil-
dren were male and 30% (n = 66) female.
Sixty-seven percent (n = 146) were cases
of pure plagiocephaly, 12% (n = 26) were
brachycephaly cases, and 21% (n = 46)
had a combination of the two.
The average degree of severity of the
plagiocephaly was a CVAI of 11.5% (me-
dian 11.1%, range 1.4–24.6%, standard
deviation (SD) 4.5%). With a mean asym-
metry of 13.8%, the helmet group had a
significantly more pronounced head defor-
mity than the group without helmets
(mean CVAI = 8.7%; P < 0.0001).
Brachycephaly was considerably preva-
lent in the group as a whole, with a mean
cranial index value of 101.7% (median
101.6%, range 80–116.53%, SD 5.8%).
The children in the helmet group showed
significantly more severe brachycephaly
(P < 0.0001).
Treatment pathways
The conspicuous head shape was first
detected during the first 4 months of
life for 78.4% of the children. The peak
incidence was at 3 months (19.7%) in
children who later received helmet ther-
apy and at 4 months (19.8%) in children
without helmet treatment. In the majority
of cases (60.8%), the abnormality was
recognized by the parents first, and in
25.9% of cases by the paediatrician.
Treatment of infant positional cranial deformity
Midwives and physiotherapists played a
lesser role, recognizing 3.5% and 3.1% of
cases, respectively, as did relatives, friends,
and strangers.
Almost all parents (n = 216, 99.1%)
asked their paediatrician about the prob-
lem. The latency between initial recogni-
tion and addressing the problem with the
paediatrician was only 0.41 months on
average (median 0.00 months). The time
lag was less than 1 month in 78.9%. The
diagnosis was made directly during the
visit to the paediatrician for 40.8% of
children. In total, there was a mean time
delay of 1.44 months (median 1.0 month)
until a definitive pathological diagnosis
was made.
In the majority of cases (62.4%), the
parents were informed that growth would
probably result in a satisfactory spontane-
ous improvement. Direct referral to a cra-
niofacial specialist occurred for only 5.3%
of children without helmet treatment and
6.6% of children with subsequent helmet
therapy. Simple measures such as active
positioning of the infant and positioning
aids (positioning pillows) were recom-
mended in over 90%. Implementation of
the methods was usually prompt.
There was a mean delay of 0.49
months (mean 0 months). More than
half the children received additional
treatment such as physiotherapy and
Table 1. Frequencies of preclinical treatments applied.
Treatment prior to first presentation at
the craniofacial clinic
None
Active positioning/positioning aids
Physiotherapy/osteopathy
Positioning and physiotherapy/osteopathy
1173
osteopathy (75.2%). The mean time from
first recognition to the implementation of
this additional therapy was 0.98 months
(median 1.0 month). Children with severe
head shape deformities and subsequent
helmet therapy received combination treat-
ment (80.3%). The sole use of positioning
or other professional measures was uncom-
mon (Table 1).
The first presentation to our clinic was
at a mean age of 5.74 months (median 5.0
months) for children with subsequent
helmet therapy and 5.8 months (median
5.5 months) for children without helmet
therapy. In the group as a whole, 28.4% of
children were seen for the first time in the
specialist clinic at >6 months of age. The
mean time delay between initial recogni-
tion of the deformity and first presentation
was 3.33 months (median 3.0 months).
Almost half (46%) had a time delay of
more than 3 months. No statistically sig-
nificant difference in the time delay was
found between children with and without
later helmet treatment (P = 0.208). In con-
trast to this, the time delay for children
who underwent physiotherapy or osteopa-
thy treatment was significantly higher
(P = 0.023). The mean delay was 3.46
months (median 3.0 months) for children
undergoing such treatments versus 2.69
months (median 2.0 months) for children
who did not.
The chronological relationship between
important care management steps and the
treatment of positioning-related deformi-
ties is shown in Fig. 4; the delays are
summarized in Table 2.
Discussion
Positional head shape deformities have
become common. The increased incidence
and exchange of information using mod-
ern media communication have increased
parent and health care provider awareness
of this problem. Given the obvious asym-
metric or disproportionate head of their
beloved child, they are often very anxious
and seek help and advice.
In addition to the possible persistence of
the deformity causing aesthetic problems,
Helmet therapy
Yes
No (n = 96) (n = 122) All (n = 218)
n % n % n %
1 1.0% 1 0.8% 2 0.9%
19 19.8% 14 11.5% 33 15.1%
10 10.4% 9 7.4% 19 8.7%
66 68.8% 98 80.3% 164 75.2%
1174 Kluba et al.
Fig. 4. Chronological course of important care management steps.
there is also the worry of potential func- or standard pathways available in the
tional disorders at a later date and this has German health system. Treatment meth-
resulted in a justifiable increase in demand ods remain individual and depend on the
for information and treatment. Long-term evaluation and opinion of the therapists
consequences such as cognitive or motor and health professionals involved. This
function developmental retardation, visual might be different in other countries.
field restriction, psychological stress such According to our data, the deformity
as bullying, and jaw asymmetry, as well as becomes noticeable at between 3 and 4
muscular problems, have been described months of age on average. A mean of 3
in various publications.7,18,19 However, months pass from initial recognition of the
many of the studies have had shortfalls disorder to first presentation at the special-
in their design, and the data should be ist clinic. This time lag is critical consid-
viewed critically. ering the fact that helmet therapy is
There is disagreement among experts, recognized as an effective method3–13
especially regarding the long-term conse- but that the efficiency of the method is
quences. The clinical relevance and the time-dependent.20 Studies therefore rec-
need for therapeutic measures are also a ommend that this treatment is started ear-
contentious issue. This is reflected in an ly, ideally between 4 and 6 months of
additional uncertainty in parents. age.6,14,15 Nevertheless, children pre-
A standardized, established strategy for sented to us for the first time at 6 months
the care of the patient is therefore desir- of age on average, and more than a quarter
able, but does not yet exist, at least not in (29.7%) even later. Keeping in mind the
Germany. To date there are no guidelines additional time needed for an individual
Table 2. Delays between important care management steps.
Delay from: Helmet (n = 122) No helmet (n = 96)
Median months (SD) Median months (SD)
Detection to paediatrician 0.0 (1.3) 0.0 (0.6)
Paediatrician to diagnosis 1.0 (1.8) 1.0 (2.3)
Detection to physiotherapy/osteopathy 1.0 (1.7) 1.0 (1.5)
Physiotherapy/osteopathy to first 3.0 (2.0) 2.0 (1.8)
craniofacial presentation
Paediatrician to first craniofacial 3.0 (2.0) 2.0 (2.0)
presentation
Detection to first craniofacial 3.0 (2.2) 3.1 (2.1)
presentation
SD, standard deviation.
helmet to be made (2 weeks), this delay of
several months denotes a potentially
poorer treatment result, at least for chil-
dren who would benefit from a helmet.
Furthermore, it should be considered that
the parents often come under increased
pressure to make a decision as a result
of the late presentation. An immediate or
rapid decision for or against helmet thera-
py is needed so that no more time is lost.
Time to reconsider or to clarify funding
can usually only be ‘bought’ with a poten-
tially poorer outcome.
Detailed observation showed that par-
ents contacted their local paediatrician in
the first instance without any relevant
delay. In over 60% of cases, the physician
consulted initially favoured spontaneous
improvement, and positioning methods,
physiotherapy, and osteopathy were wide-
ly accepted in the treatment of positional
head deformities. They were recom-
mended by the majority and were imple-
mented with very little delay. In contrast
to this, helmet therapy appears not to be so
commonly used or so favoured. Only a
few children were referred directly to the
specialist clinic. Established methods
were used initially, and only cases with
a lack of success or success failure were
referred. The significantly later presenta-
tion of children receiving physiotherapy or
osteopathy treatment in the specialist clin-
ic confirmed this.
We therefore see potential for optimiz-
ing the treatment pathway at this point. It
is our opinion that affected children should
at best be referred immediately to a spe-
cialist once the problem is identified, irre-
spective of the person who has seen the
child primarily (paediatrician, therapist).
To facilitate the identification of problem-
atic positional cranial deformities, we also
suggest that the measurement of cranial
diameters becomes an obligatory part of
the paediatric well-child visits between
the third and fourth months of life.
In principal, a stepwise therapeutic ap-
proach (positioning methods ! phy-
physiotherapy or osteopathy ! helmet
therapy) appears sensible. However, the
period of time between first recognition
and the time limit for an optimal start of
helmet therapy is very short. It is also too
short to be able to reliably assess the
therapeutic effects of the initial methods
chosen. Furthermore a stepwise approach
delays referral to a specialist and therefore
would potentially be disadvantageous for
children with an indication for helmet
therapy. Time delays can also promote
uncertainty or a later reproachful attitude
in the parents of these children. The chal-
lenge is to reliably and promptly identify

these children. They should therefore at-
tend a specialist clinic or practice as soon
as possible, regardless of whether the first
physician consulted is for or against hel-
met therapy. In cases of necessity, the
chance of a prompt initiation of helmet
therapy would therefore not be missed and
this concept can be carried out alongside
other treatments. Physiotherapy and oste-
opathy treatments can be performed at the
same time as helmet therapy. The time
delay for children with an indication for
helmet therapy would thereby be mini-
mized and pressure would be taken off
the parents to make a direct decision about
the treatment.
In addition, the focus of clinical and
scientific efforts should be directed more
towards raising parental awareness of the
subject earlier in terms of prophylaxis.
When suitable positioning aids, for exam-
ple, are used from birth onwards, the
development of a head deformity that
requires treatment can be counteracted
or the severity can be reduced.
The treatment of positional head defor-
mities is an interdisciplinary and interpro-
fessional challenge. The paediatrician, as
the person contacted first and without any
relevant delay, has a key role. Watchful
waiting and physiotherapy or osteopathy
are suitable treatment approaches. How-
ever, clarification of whether there is an
indication for helmet therapy should take
place early. A delay in starting helmet
therapy to >6 months of age can result
in a poorer treatment outcome for these
children. The present study shows that up
to now, there has been a delay of several
months between initial recognition of the
deformity and first presentation to the
specialist clinic. In order to avoid such a
delay, prompt referral to a specialist insti-
tution and, when necessary, the simulta-
neous use of various treatments should be
the preferred concept in the management
of positional cranial deformities.
Funding
None.
Competing interests
None declared.
Ethical approval
Approval was obtained from the Ethics
Board of the Medical Faculty of the Uni-
versity Tübingen (696/2012BO2).
Treatment of infant positional cranial deformity
Patient consent
Consent was obtained.
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Address:
Susanne Kluba
Department of Oral and Maxillofacial
Surgery
University Hospital Tübingen
Osianderstr. 2–8
D-72076 Tübingen
Germany
Tel.: +49 7071 2986174;
Fax: +49 7071 2983481.
E-mail: susanne.kluba@med.uni-tuebin-
gen.de